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LETTER TO EDITOR  
Year : 2016  |  Volume : 59  |  Issue : 3  |  Page : 431-433
Salivary gland anlage tumor


Department of Pathology, Max Super Speciality Hospital, New Delhi, India

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Date of Web Publication10-Aug-2016
 

How to cite this article:
Khurana A, Singaravel S, Mukherjee U. Salivary gland anlage tumor. Indian J Pathol Microbiol 2016;59:431-3

How to cite this URL:
Khurana A, Singaravel S, Mukherjee U. Salivary gland anlage tumor. Indian J Pathol Microbiol [serial online] 2016 [cited 2019 Aug 17];59:431-3. Available from: http://www.ijpmonline.org/text.asp?2016/59/3/431/188147


Editor,

Upper respiratory tract obstruction may be life-threatening in neonates as they are obligate nasal breathers.

We present the case of a 3-month-old male infant brought with complaints of difficulty in breathing and feeding since birth. Otorhinolaryngological examination showed an erythematous midline nasopharyngeal mass. On noncontrast computerized tomography (CT), a lobulated soft-tissue mass was identified arising from the posterior wall of the nasopharynx, causing partial obstruction of the airway [Figure 1]a and [Figure 1]b. Multiplanar magnetic resonance imaging of the nasopharynx delineated the mass, estimating it to be 11 mm × 8 mm in size, attached to the posterosuperior wall by a narrow pedicle. The mass was excised under endoscopic guidance and sent for histopathological examination.
Figure 1: (a) Sagittal plane and (b) transverse plane noncontrast computerized tomography image showing the soft-tissue mass (indicated by arrowhead) arising in the posterior wall of the nasopharynx, partially obstructing the airway. (c) Lining epithelium and nodular tumor mass underneath (H and E, ×40). (d) Tumor with biphasic proliferation of epithelial/ductal cells and spindle cells (H and E, ×400). (e) CK AE1/AE3 ×400 highlighting the epithelial cell population. (f) Vimentin ×400 highlighting the spindle cell component

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The gross specimen consisted of a single, irregular mucosa covered soft-tissue mass measuring 1 cm × 0.8 cm × 0.5 cm which was bisected and processed entirely. Microscopic examination showed surface lining of nonkeratinized stratified squamous epithelium ulcerated by a polypoid tumor with multinodular growth pattern [Figure 1]c. The tumor showed a biphasic proliferation of bland-appearing epithelial/ductal units merging with cellular nodules and fascicles of monomorphic spindle cells [Figure 1]d. The latter showed mitotic activity. No necrosis or anaplasia was seen. Features were suggestive of a benign biphasic neoplasm likely of salivary gland origin, and immunohistochemistry (IHC) was performed. The epithelial cell population was highlighted by CK [Figure 1]e and EMA. Vimentin [Figure 1]f and SMA highlighted the background stromal cell population. In view of the histomorphological features and IHC findings, a diagnosis of salivary gland anlage tumor (SGAT) was rendered. The postoperative course was uneventful, and at 2-year follow-up, the child was symptom-free.

Nasopharyngeal neoplasms are an unusual cause of nasal obstruction in neonates.[1] SGAT, also referred to as congenital pleomorphic adenoma, is an extremely rare congenital tumor of the nasopharynx. First characterized by Dehner et al. in 1994,[2] approximately 24 cases have been reported in world literature.[3] It has been hypothesized that SGAT is probably a hamartoma of minor salivary gland derivation due to its midline location and benign behavior. There is a male preponderance and most cases present in the first 2 weeks of life with nasal obstruction worsened during feeding with improvement on crying.[1]

As the nasopharynx is difficult to access, multiple diagnostic approaches may be required for visualization including anterior rhinoscopy, fiber-optic endoscopy, and radiological modalities such as CT and magnetic resonance imaging. SGATs are mucosa-covered polypoid lesions attached to the posterior wall of the pharynx by a thin, delicate vascular pedicle. There is no risk of malignancy; however, microscopic features such as necrosis, squamous cell metaplasia, cystic degeneration,[4] and high mitotic index [5] may be present, mimicking a more ominous lesion. The important differentials in this age group include developmental masses such as basal cephalocele, nasal glioma, and nasolacrimal mucocele, and benign neoplasms such as dermoid, hamartoma, teratoma, and hemangioma which can be excluded on microscopic examination. Malignant neoplasms of this region such as neuroblastoma, Langerhans cell histiocytosis, chloroma, rhabdomyosarcoma, and lymphoma occur in older children and are rare in the neonatal period.[6] Simple excision is curative, but complications may arise including torrential bleeding from the pedicle which may be torn by probing or instrumentation; dislodgment of the mass may result in fatal airway obstruction.[1]

To conclude, we report a case of SGAT, which is a very rare cause of respiratory distress in the neonatal period. Due to its rarity, it may remain unsuspected by the clinician and pathologist. It may mimic a malignancy on histopathology. Finally, in spite of the benign biological behavior, tearing of the pedicle may result in life-threatening complications such as bleeding or airway obstruction by the dislodged mass.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Cohen EG, Yoder M, Thomas RM, Salerno D, Isaacson G. Congenital salivary gland anlage tumor of the nasopharynx. Pediatrics 2003;112 (1 Pt 1):e66-9.  Back to cited text no. 1
    
2.
Dehner LP, Valbuena L, Perez-Atayde A, Reddick RL, Askin FB, Rosai J. Salivary gland anlage tumor (“congenital pleomorphic adenoma”). A clinicopathologic, immunohistochemical and ultrastructural study of nine cases. Am J Surg Pathol 1994;18:25-36.  Back to cited text no. 2
    
3.
Tinsa F, Boussetta K, Bousnina S, Menif K, Nouira F, Haouet S, et al. Congenital salivary gland anlage tumor of the nasopharynx. Fetal Pediatr Pathol 2010;29:323-9.  Back to cited text no. 3
    
4.
Michal M, Sokol L, Mukensnabl P. Salivary gland anlage tumor. A case with widespread necrosis and large cyst formation. Pathology 1996;28:128-30.  Back to cited text no. 4
    
5.
Marien A, Maris M, Verbeke S, Creytens D, Verlooy J, Van Reempts P, et al. An unusual tumour causing neonatal respiratory distress. B-ENT 2012;8:149-51.  Back to cited text no. 5
    
6.
Mogensen MA, Lin AC, Chang KW, Berry GJ, Barnes PD, Fischbein NJ. Salivary gland anlage tumor in a neonate presenting with respiratory distress: Radiographic and pathologic correlation. AJNR Am J Neuroradiol 2009;30:1022-3.  Back to cited text no. 6
    

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Correspondence Address:
Dr. Saranya Singaravel
11/1 Sagar Tarang, Khan Abdul Gaffar Khan Road, Worli Sea Face, Mumbai - 400 030, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.188147

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